We present the case of an 11-year-old boy with humoral immunodeficiency on monthly intravenous immunoglobulins (IVIG) infusions, evaluated for recurrent, brief, neurological deficits secondary to cerebral sinus thrombosis without any identifiable hypercoagulability state. Etiologic possibilities for the thrombotic event are presented with special discussion of IVIG-related cerebral thrombosis. To the best of our knowledge, our patient represents the first reported case of Bruton's disease on IVIG therapy developing a cerebral ischemic event and the second reported case of cerebral sinus thrombosis associated with IVIG use for any disease. Potential concerns regarding the risk of cerebral thrombosis during IVIG therapy in this and other disorders are reviewed.